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Yusuke Nishimura, Atsushi Natsume, and Howard J. Ginsberg

The authors describe a case of a 79-year-old man with a lumbar spinal dural arteriovenous fistula (DAVF) and isthmic spondylolisthesis at the same level. The patient's thoracic spine MRI study demonstrated swelling and increased T2 signal in the spinal cord and regional dilated perimedullary vessels. Lumbar spine MRI showed L-4 isthmic spondylolisthesis with severe bilateral L4–5 foraminal stenoses. Spinal angiography revealed a fistulous connection at the left L-4 nerve root sleeve between perimedullary veins and a dural branch of the L-4 radicular artery. Based on previous reports about secondary spinal DAVFs, the abnormal vascular communication likely developed secondary to the microtrauma and inflammation on the left L-4 nerve root sleeve, which was attributable to the isthmic spondylolisthesis. The authors performed disconnection of the arteriovenous shunt as well as an L4–5 decompression and posterior instrumented fusion with pedicle screws. The patient's postoperative course was uneventful, and he improved neurologically. It is important to bear in mind that a spinal DAVF may develop as a consequence of any sort of trauma or inflammation involving nerve roots. One should consider the concomitant treatment of both the spinal DAVF and the underlying pathology that may have given rise to the spinal DAVF.

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Kuniaki Tanahashi, Kenji Uda, Yoshio Araki, Kazuhito Takeuchi, Jungsu Choo, Lushun Chalise, Kazuya Motomura, Fumiharu Ohka, Toshihiko Wakabayashi, and Atsushi Natsume

The presigmoid approach (PSA) is selected to obtain more lateral access to cerebellopontine angle tumors, brainstem cavernous malformations, or vertebrobasilar artery aneurysms than the standard retrosigmoid approach. However, mastoidectomy for the PSA can be considered time-consuming and to carry a higher risk of complications due to the anatomical complexity of the region. The authors established a method of minimized mastoidectomy focused on exposing Trautmann’s triangle as the corridor for the PSA while maximizing procedural simplicity and safety and maintaining a sufficient operative view. The authors present their method of minimized mastoidectomy in a cadaver dissection and operative cases, showing potential as a useful option for the PSA.

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Toru Arima, Atsushi Natsume, Hisashi Hatano, Norimoto Nakahara, Mitsugu Fujita, Dai Ishii, Toshihiko Wakabayashi, Manabu Doyu, Tetsuro Nagasaka, and Jun Yoshida

✓ A rare case of chordoid meningioma in the lateral ventricle observed in an adult is reported. The first clinical manifestation of the disease was a prolonged fever of unknown origin. Abnormalities in the patient's blood chemistry, principally polyclonal hypergammaglobulinemia (immunoglobulin [Ig]G, IgA, and markedly IgE) and an elevated serum level of C-reactive protein, were associated with the disease. The tumor was histologically confirmed to be a chordoid meningioma, and its surgical removal resulted in complete resolution of the patient's symptoms. By combining reverse transcription—polymerase chain reaction and immunohistochemical analysis, it may be shown that cytokine production, including that of interleukin (IL)-6, IL-1β, and vascular endothelial growth factor, plays a role in the pathogenesis of chordoid meningioma associated with Castleman syndrome.

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Kazuya Motomura, Masazumi Fujii, Satoshi Maesawa, Shunichiro Kuramitsu, Atsushi Natsume, and Toshihiko Wakabayashi

Alexia and agraphia are disorders common to the left inferior parietal lobule, including the angular and supramarginal gyri. However, it is still unclear how these cortical regions interact with other cortical sites and what the most important white matter tracts are in relation to reading and writing processes.

Here, the authors present the case of a patient who underwent an awake craniotomy for a left inferior parietal lobule glioma using direct cortical and subcortical electrostimulation. The use of subcortical stimulation allowed identification of the specific white matter tracts associated with reading and writing. These tracts were found as portions of the dorsal inferior frontooccipital fasciculus (IFOF) fibers in the deep parietal lobe that are responsible for connecting the frontal lobe to the superior parietal lobule. These findings are consistent with previous diffusion tensor imaging tractography and functional MRI studies, which suggest that the IFOF may play a role in the reading and writing processes. This is the first report of transient alexia and agraphia elicited through intraoperative direct subcortical electrostimulation, and the findings support the crucial role of the IFOF in reading and writing.

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Kenichiro Iwami, Hiroyuki Momota, Atsushi Natsume, Sayano Kinjo, Tetsuya Nagatani, and Toshihiko Wakabayashi

Object

Mouse models have been widely used in developing therapies for human brain tumors. However, surgical techniques such as bone drilling and skin suturing to create brain tumors in adult mice are still complicated. The aim of this study was to establish a simple and accurate method for intracranial injection of cells or other materials into mice.

Methods

The authors performed micro CT scans and skull dissection to assess the anatomical characteristics of the mouse postglenoid foramen. They then used xenograft and genetically engineered mouse models to evaluate a novel technique of percutaneous intracranial injection via the postglenoid foramen. They injected green fluorescent protein–labeled U87MG cells or virus-producing cells into adult mouse brains via the postglenoid foramen and identified the location of the created tumors by using bioluminescence imaging and histological analysis.

Results

The postglenoid foramen was found to be a well-conserved anatomical structure that allows percutaneous injection into the cerebrum, cerebellum, brainstem, and basal cistern in mice. The mean (± SD) time for the postglenoid foramen injection technique was 88 ± 15 seconds. The incidence of in-target tumor formation in the xenograft model ranged from 80% to 100%, depending on the target site. High-grade gliomas were successfully developed by postglenoid foramen injection in the adult genetically engineered mouse using virus-mediated platelet-derived growth factor B gene transfer. There were no procedure-related complications.

Conclusions

The postglenoid foramen can be used as a needle entry site into the brain of the adult mouse. Postglenoid foramen injection is a less invasive, safe, precise, and rapid method of implanting cells into the adult mouse brain. This method can be applied to both orthotopic xenograft and genetically engineered mouse models and may have further applications in mice for the development of therapies for human brain tumors.

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Kuniaki Tanahashi, Kenji Uda, Yoshio Araki, Kazuhito Takeuchi, Jungsu Choo, Lushun Chalise, Kazuya Motomura, Fumiharu Ohka, Toshihiko Wakabayashi, and Atsushi Natsume

The presigmoid approach (PSA) is selected to obtain more lateral access to cerebellopontine angle tumors, brainstem cavernous malformations, or vertebrobasilar artery aneurysms than the standard retrosigmoid approach. However, mastoidectomy for the PSA can be considered time-consuming and to carry a higher risk of complications due to the anatomical complexity of the region. The authors established a method of minimized mastoidectomy focused on exposing Trautmann’s triangle as the corridor for the PSA while maximizing procedural simplicity and safety and maintaining a sufficient operative view. The authors present their method of minimized mastoidectomy in a cadaver dissection and operative cases, showing potential as a useful option for the PSA.

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Kazuya Motomura, Lushun Chalise, Fumiharu Ohka, Kosuke Aoki, Kuniaki Tanahashi, Masaki Hirano, Tomohide Nishikawa, Junya Yamaguchi, Hiroyuki Shimizu, Toshihiko Wakabayashi, and Atsushi Natsume

OBJECTIVE

Lower-grade gliomas (LGGs) are often observed within eloquent regions, which indicates that tumor resection in these areas carries a potential risk for neurological disturbances, such as motor deficit, language disorder, and/or neurocognitive impairments. Some patients with frontal tumors exhibit severe impairments of neurocognitive function, including working memory and spatial awareness, after tumor removal. The aim of this study was to investigate neurocognitive and functional outcomes of frontal LGGs in both the dominant and nondominant hemispheres after awake brain mapping.

METHODS

Data from 50 consecutive patients with diffuse frontal LGGs in the dominant and nondominant hemispheres who underwent awake brain surgery between December 2012 and September 2018 were retrospectively analyzed. The goal was to map neurocognitive functions such as working memory by using working memory tasks, including digit span testing and N-back tasks.

RESULTS

Due to awake language mapping, the frontal aslant tract was frequently identified as a functional boundary in patients with left superior frontal gyrus tumors (76.5%). Furthermore, functional boundaries were identified while evaluating verbal and spatial working memory function by stimulating the dorsolateral prefrontal cortex using the digit span and visual N-back tasks in patients with right superior frontal gyrus tumors (7.1%). Comparing the preoperative and postoperative neuropsychological assessments from the Wechsler Adult Intelligence Scale–Third Edition (WAIS-III) and Wechsler Memory Scale–Revised (WMS-R), significant improvement following awake surgery was observed in mean Perceptual Organization (Z = −2.09, p = 0.04) in WAIS-III scores. Postoperative mean WMS-R scores for Visual Memory (Z = −2.12, p = 0.03) and Delayed Recall (Z = −1.98, p = 0.04) were significantly improved compared with preoperative values for every test after awake surgery. No significant deterioration was noted with regard to neurocognitive functions in a comprehensive neuropsychological test battery. In the postoperative course, early transient speech and motor disturbances were observed in 30.0% and 28.0% of patients, respectively. In contrast, late permanent speech and motor disturbances were observed in 0% and 4.0%, respectively.

CONCLUSIONS

It is noteworthy that no significant postoperative deterioration was identified compared with preoperative status in a comprehensive neuropsychological assessment. The results demonstrated that awake functional mapping enabled favorable neurocognitive and functional outcomes after surgery in patients with diffuse frontal LGGs.

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Yusuke Nishimura, Masahito Hara, Atsushi Natsume, Yasuhiro Nakajima, Ryuichi Fukuyama, Toshihiko Wakabayashi, and Howard J. Ginsberg

A spinal intradural extramedullary venous angioma is extremely rare and has not been previously reported. In this paper, the authors report on this entity with morphological and immunohistochemical evidence, and discuss the surgical strategy for its treatment. A 54-year-old woman presented to Nagoya University Hospital complaining of left-sided pain in the hip, thigh, and inguinal and perianal regions, with progressive worsening during the previous 2 weeks. Lumbar spine MRI showed an intradural extramedullary cyst at the level of T12–L1, which extended from the conus medullaris to the cauda equina. The cyst wall was not enhanced on T1-weighted MRI with Gd. Intraoperatively, a midline dural opening allowed the authors to easily visualize a dark-reddish cyst behind the spinal nerve rootlets in the cauda equina adjacent to the conus medullaris. The cyst was believed to originate from one of the spinal nerve rootlets in the cauda equina and a cluster of veins was identified on the cyst wall. The cyst was resected with the affected nerve rootlet. The surgery left no detectable neurological deficit. Based on the morphological and immunohistochemical evidence, the lesion was diagnosed as a venous angioma. No tumor recurrence was confirmed based on MRI at the time of the 2-year follow up. This is the first report of an intradural extramedullary cystic venous angioma that was successfully resected.

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Kazuya Motomura, Lushun Chalise, Hiroyuki Shimizu, Junya Yamaguchi, Tomohide Nishikawa, Fumiharu Ohka, Kosuke Aoki, Kuniaki Tanahashi, Masaki Hirano, Toshihiko Wakabayashi, and Atsushi Natsume

OBJECTIVE

This study aimed to evaluate the efficacy of levetiracetam (LEV) combined with perampanel (PER) therapy for intraoperative seizure treatment to determine whether a combination of LEV and PER can aid in the prevention of intraoperative intractable seizures during awake surgery.

METHODS

The authors performed a retrospective cohort study in 78 consecutive patients with glioma who underwent awake surgery using intraoperative direct electrical stimulation mapping. To prevent intraoperative seizures, 50 patients were treated with the antiepileptic drug LEV only (LEV group) from January 2017 to January 2019, while the remaining 28 patients were treated with LEV plus PER (LEV + PER group) between March 2019 and January 2020. LEV (1000–3000 mg) and/or PER (2–4 mg) were administered before the surgery.

RESULTS

Preoperative seizures with International League Against Epilepsy (ILAE) class II–VI occurred in 44% of the patients in the LEV group and in 35.7% of patients in the LEV + PER group, with no significant difference between groups (p = 0.319). Total intraoperative seizures occurred in 18 patients (36.0%) in the LEV therapy group and in 2 patients (7.1%) in the LEV + PER group (p = 0.009). Of these, there were no patients (0%) with intractable seizures in the LEV + PER group. Regarding factors that influence intraoperative seizures in glioma patients during awake brain surgery, multivariate logistic regression models revealed that the occurrence of intraoperative seizures was significantly related to the involvement of motor-related regions (positive vs negative, HR 6.98, 95% CI 1.71–28.56, p = 0.007), preoperative seizure (ILAE class II–VI vs ILAE class I, HR 4.44, 95% CI 1.22–16.11, p = 0.024), and LEV + PER group (positive vs negative, HR 0.07, 95% CI 0.01–0.44, p = 0.005). Treatment-related adverse effects were rare and mild, including sleepiness, tiredness, and dizziness in both treatment groups.

CONCLUSIONS

This study demonstrates that LEV + PER therapy is significantly associated with a lower risk of intraoperative seizures compared with LEV therapy alone in patients with glioma during awake brain mapping. These findings will help neurosurgeons conduct safe and reliable awake surgeries and reduce the rate of intraoperative intractable seizures during such procedures.

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Hiroyuki Shimizu, Kazuya Motomura, Fumiharu Ohka, Kosuke Aoki, Kuniaki Tanahashi, Masaki Hirano, Lushun Chalise, Tomohide Nishikawa, Junya Yamaguchi, Jun Yoshida, Atsushi Natsume, and Toshihiko Wakabayashi

OBJECTIVE

The current study aimed to evaluate the treatment outcomes and toxicities of patients with intracranial germ cell tumors (GCTs).

METHODS

This study retrospectively included 110 consecutive patients (70 patients in the germinomatous group and 40 patients in the nongerminomatous GCT [NGGCT] groups) receiving surgery, platinum-based chemotherapy, and radiotherapy for newly diagnosed primary intracranial GCTs. In the authors’ protocol, patients with GCTs were further divided into the following four groups: the germinomatous group and the NGGCT groups (mature teratoma, intermediate prognosis, or poor prognosis).

RESULTS

The median overall survival (OS) and progression-free survival (PFS) rates of the patients in the germinomatous group were significantly higher than those in the NGGCT group (p < 0.001). The 5-, 10-, and 20-year OS rates in the germinomatous group were 97.1%, 95.7%, and 93.2%, respectively, with a median follow-up of 11.0 years. On the contrary, the 5-, 10-, and 20-year OS rates in the NGGCT group were 67.3%, 63.4%, and 55.4%, respectively. The 5-, 10-, and 20-year PFS rates were 91.4%, 86.6%, and 86.6%, respectively, in the germinomatous group, whereas those of the NGGCT group were approximately 67.4%, 60.2%, and 53.5%, respectively. Based on the four types of classification in our study, the 5-, 10-, and 20-year OS rates in the NGGCT intermediate prognosis group were 78.9%, 71.8%, and 53.8%, respectively. On the contrary, the 3- and 5-year OS rates in the NGGCT poor prognosis group were 42.9% and 34.3%, respectively. Moreover, toxicities with the treatment of intracranial GCTs were found to be tolerable in the present study population. The multivariate survival models for OS in the NGGCT intermediate prognosis and poor prognosis groups demonstrated that only the alpha-fetoprotein status was significantly associated with worsened OS (HR 3.88, 95% CI 1.29–11.66; p = 0.02).

CONCLUSIONS

The authors found that platinum-based chemotherapy and radiotherapy result in favorable survival outcomes in patients with germinomatous GCTs. Clinical outcomes were still unfavorable in the NGGCT intermediate prognosis and poor prognosis groups; therefore, a new protocol that increases the survival rate of patients belonging in both groups should be considered.